J Oral Med Pain.  2023 Jun;48(2):69-73. 10.14476/jomp.2023.48.2.69.

Temporomandibular Disorders Symptoms Associated with Multicentric Reticulohistiocytosis: A Case Report

• Jun S
• Park JW

• KMID: 2552142
• DOI: http://doi.org/10.14476/jomp.2023.48.2.69

Abstract

This report describes the case of a 58-year-old woman who visited our outpatient clinic with the chief complaint of temporomandibular disorders (TMD) related symptoms and later reported a previous diagnosis of multicentric reticulohistiocytosis (MRH). The patient had a history of leg edema, pain, and skin lesions on the hands and knees, which were diagnosed as MRH by tissue biopsy. Temporomandibular joint pain and mouth-opening limitation developed 1 year ago. Radiographs of the maxilla and mandible revealed severe resorption of the mandibular condyle on both sides, resulting in the complete loss of the original shape of the condyle. The articular fossa was also affected, and consequently, the joint space was excessively widened. MRH is a rare non-Langerhans cell histiocytosis that primarily affects the skin and joints, causing skin nodules and joint pain along with bony deformity. Reports of involvement of tissues or organs other than the skin and joints of the extremities are limited, and to our knowledge, only one case of MRH with dental manifestation has been reported. Thus, this case report highlights the importance of considering MRH as a possible diagnosis in patients with TMD symptoms, particularly in those accompanied by severe bone resorption.

Keyword

Bone resorption; Histocytosis; Histiocytosis, Non-Langerhans-cell; Multicentric reticulohistiocytosis; Temporomandibular disorders